Deep-seated glands as result of so-called epithelial misplacement are a well-recognised entity in the large bowel in association with large, usually villous adenomas following repeated biopsies or partial excision. A similar phenomenon is encountered with in the small intestine hamartomatous polyps in patients with Peutz-Jegher syndrome. Here, the accepted mechanism is thought to be trauma from torsion of these pedunculated polyps, which induces a mechanical disruption at the base of the adenoma resulting in the deeper placed glands being forced into the submucosa. An investing cuff of normal lamina propria usually surrounds these misplaced glands, and there is haemorrhage, haemosiderin and fibrosis in the general vicinity of the glands. Another cause of deep-seated benign glands in the colon is colitis cystica profunda, a condition typically associated with rectal mucosal prolapse, inflammatory bowel disease and radiation treatment. A similar lesion termed “gastritis cystica profunda” is seen in the stomach several years after surgery with ischaemia and chronic inflammation thought to be instrumental. The glands are typically located in the submucosa.
The case presented here describes a gastric foveolar-type adenoma, which after three previous endoscopic attempts at removal, showed after definitive gastric sleeve resection, misplaced gastric glands deep within the muscularis propria.
Click on the below thumbnails to view more information:
In 1947 Scott and Payne first described cystic, dilated gastric glands in the submucosa and thought the presence of glands in this location represented a congenital abnormality [1]. Oberman and colleagues were of a similar opinion and construed glands located in the submucosa to be heterotopic in aetiology [2]. Several years later in 1972, Littler and Glibermann suggested the presence of cystically dilated gastric glands in the submucosa was a reactive and post-surgical condition (at gastro-enterostomy sites) and they coined the term, “gastritis cystica polyposa” which subsequently became “gastritis cystica profunda” (GCP) as the process in the stomach recapitulated a similar to that occurring in the colon [3]. The accepted pathogenesis is thought to be related to several factors working in concert: surgery predisposing to defects in the mucosa, chronic ischaemia and inflammation all allowing for mucosal prolapse and herniation of glands into the submucosa. The interval between surgery and the development of GCP has a wide range with an interval from 3 to 40 years post-surgery being quoted by Franzin et al [4]. The strong association of GCP with gastric cancer, especially in the early literature is intriguing [1]. In the series reported by Watanabe et al, 3 of 9 cases of GCP had coexistent adenocarcinoma [5]. Several have postulated that the cystically dilated and hyperplastic glands have an increased propensity and risk for malignancy and are thus pre-malignant. However, it is conjectural whether the submucosal cystic glands ever develop dysplasia and subsequent carcinoma. The fact that the initial polyp in the case presented herein contained foci of low-grade dysplasia is coincidental as the deep-lying glands were completely benign. The incidence and occurrence of dysplasia is more than likely related to the polyp rather than GCP.
This case departs from all previous reported cases of GCP because of the presence of benign, cystically dilated glands within the muscularis propria. This degree and depth of extension of GCP is extremely rare and as such mimics infiltrating adenocarcinoma. However, the architecture (uniformity of cystically dilated glands, lack of angulated contours, a non-infiltrative pattern, absence of stromal desmoplasia, a surrounding cuff of lamina propria and lack of disruption of the muscle fibres) and bland, benign cytological features, all point against an infiltrating adenocarcinoma.
The most plausible explanation for the occurrence of the glands within the muscularis propria is related to the three previous endoscopic manipulations in which attempts were made to remove as much of the polyp as possible. Electro-cautery of the base of the polyp would have created a defect with thinning, weakening and obliteration of the submucosa underlying the polyp. This in turn results in apposition of the base of the polyp to the muscularis propria and with further endoscopic manipulation facilitated herniation and prolapse of the deep-lying glands of the polyp into the muscularis propria. This process is analogous to the iatrogenically induced artifactual endomyometrial clefts and tears leading to misplaced endometrial tissue, resembling “adenomyosis” following laparoscopic hysterectomy and transcervical resection of the endometrium (TCRE) [6].
This is a rare case of GCP in which the misplaced glands were located deep within the fibres of the muscularis propria. Multiple prior endoscopic manipulations facilitated herniation and prolapse through defects in the gastric wall. These deeply located glands should not be mistaken for invasive adenocarcinoma. Careful attention to the absence of an invasive growth pattern, lack of stromal desmoplasia (with retention of cuffs of lamina propria around the glands) and lack of cytological atypia together with the history of multiple procedures help prevent a potential misdiagnosis.
References
- Scott H. W. and T. P. B. Payne. Diffuse congenital cystic hyperplasia of stomach clinically simulating carcinoma; report of a case. Bull Johns Hopkins Hosp 1947;81:448-55.
- Oberman HA, Lodmell CJG, Sower CND. Diffuse heterotopic cystic malformation of the stomach. N Engl J Med 1963;269:909-11.
- Littler ER, Gleibermann E. Gastritis cystica polyposa. (Gastric mucosal prolapse at gastroenterostomy site, with cystic and infiltrative epithelial hyperplasia). Cancer 1972;29:205-9.
- Franzin G, Novelli P. Gastritis cystica profunda. Histopathology 1981;5:535-47.
- Watanabe S, Tohyama T, Inagaki H, et al. Gastritis cystica profunda. Histopathological study of nine cases. Jap J Clin Oncol 1979;9:79-84.
- Krizova A, Clarke BA, Bernardini MQ, et al. Histologic artifacts in abdominal, vaginal, laparoscopic, and robotic hysterectomy specimens: a blinded, retrospective review. Am J Surg Pathol 2011;35:115-26.
